Literature DB >> 28620870

Severe growth deficiency, microcephaly, intellectual disability, and characteristic facial features are due to a homozygous QARS mutation.

Esther Leshinsky-Silver1,2,3, Jiqiang Ling4, Jiang Wu4, Chana Vinkler2,5, Keren Yosovich1, Sarit Bahar1, Miri Yanoov-Sharav2,5, Tally Lerman-Sagie2,3,6, Dorit Lev7,8,9.   

Abstract

Glutaminyl tRNA synthase is highly expressed in the developing fetal human brain. Mutations in the glutaminyl-tRNA synthetase (QARS) gene have been reported in patients with progressive microcephaly, cerebral-cerebellar atrophy, and intractable seizures. We have previously reported a new recessive syndrome of severe linear growth retardation, poor weight gain, microcephaly, characteristic facial features, cutaneous syndactyly of the toes, high myopia, and intellectual disability in two sisters of Ashkenazi-Jewish origin (Eur J Med Genet 2014;57(6):288-92). Homozygosity mapping and whole exome sequencing revealed a homozygous missense (V476I) mutation in the QARS gene, located in the catalytic domain. The patient's fibroblasts demonstrated markedly reduced QARS amino acylation activity in vitro. Furthermore, the same homozygous mutation was found in an unrelated girl of Ashkenazi origin with the same phenotype. The clinical presentation of our patients differs from the original QARS-associated syndrome in the severe postnatal growth failure, absence of epilepsy, and minor MRI findings, thus further expanding the phenotypic spectrum of the glutaminyl-tRNA synthetase deficiency syndromes.

Entities:  

Keywords:  Ashkenazi-Jewish; Growth failure; Intellectual disability; Microcephaly; QARS

Mesh:

Substances:

Year:  2017        PMID: 28620870     DOI: 10.1007/s10048-017-0516-6

Source DB:  PubMed          Journal:  Neurogenetics        ISSN: 1364-6745            Impact factor:   2.660


  8 in total

1.  Mutations in QARS, encoding glutaminyl-tRNA synthetase, cause progressive microcephaly, cerebral-cerebellar atrophy, and intractable seizures.

Authors:  Xiaochang Zhang; Jiqiang Ling; Giulia Barcia; Lili Jing; Jiang Wu; Brenda J Barry; Ganeshwaran H Mochida; R Sean Hill; Jill M Weimer; Quinn Stein; Annapurna Poduri; Jennifer N Partlow; Dorothée Ville; Olivier Dulac; Tim W Yu; Anh-Thu N Lam; Sarah Servattalab; Jacqueline Rodriguez; Nathalie Boddaert; Arnold Munnich; Laurence Colleaux; Leonard I Zon; Dieter Söll; Christopher A Walsh; Rima Nabbout
Journal:  Am J Hum Genet       Date:  2014-03-20       Impact factor: 11.025

2.  Expansion of the QARS deficiency phenotype with report of a family with isolated supratentorial brain abnormalities.

Authors:  Ramona Salvarinova; Cynthia X Ye; Andrea Rossi; Roberta Biancheri; Elke H Roland; Paul Pavlidis; Colin J Ross; Maja Tarailo-Graovac; Wyeth W Wasserman; Clara D M van Karnebeek
Journal:  Neurogenetics       Date:  2014-11-30       Impact factor: 2.660

3.  Congenital Visual Impairment and Progressive Microcephaly Due to Lysyl-Transfer Ribonucleic Acid (RNA) Synthetase (KARS) Mutations: The Expanding Phenotype of Aminoacyl-Transfer RNA Synthetase Mutations in Human Disease.

Authors:  Hugh J McMillan; Peter Humphreys; Amanda Smith; Jeremy Schwartzentruber; Pranesh Chakraborty; Dennis E Bulman; Chandree L Beaulieu; Jacek Majewski; Kym M Boycott; Michael T Geraghty
Journal:  J Child Neurol       Date:  2014-10-20       Impact factor: 1.987

4.  Progressive microcephaly is caused by compound-heterozygous mutations in QARS.

Authors:  S Waltl
Journal:  Clin Genet       Date:  2014-08-15       Impact factor: 4.438

5.  Mutations in the glutaminyl-tRNA synthetase gene cause early-onset epileptic encephalopathy.

Authors:  Hirofumi Kodera; Hitoshi Osaka; Mizue Iai; Noriko Aida; Akio Yamashita; Yoshinori Tsurusaki; Mitsuko Nakashima; Noriko Miyake; Hirotomo Saitsu; Naomichi Matsumoto
Journal:  J Hum Genet       Date:  2014-12-04       Impact factor: 3.172

6.  Mitochondrial phenylalanyl-tRNA synthetase mutations underlie fatal infantile Alpers encephalopathy.

Authors:  Jenni M Elo; Srujana S Yadavalli; Liliya Euro; Pirjo Isohanni; Alexandra Götz; Christopher J Carroll; Leena Valanne; Fowzan S Alkuraya; Johanna Uusimaa; Anders Paetau; Eric M Caruso; Helena Pihko; Michael Ibba; Henna Tyynismaa; Anu Suomalainen
Journal:  Hum Mol Genet       Date:  2012-07-23       Impact factor: 6.150

7.  A newly recognized syndrome of severe growth deficiency, microcephaly, intellectual disability, and characteristic facial features.

Authors:  Chana Vinkler; Esther Leshinsky-Silver; Marina Michelson; Dorothea Haas; Tally Lerman-Sagie; Dorit Lev
Journal:  Eur J Med Genet       Date:  2014-04-05       Impact factor: 2.708

Review 8.  The Mitochondrial Aminoacyl tRNA Synthetases: Genes and Syndromes.

Authors:  Daria Diodato; Daniele Ghezzi; Valeria Tiranti
Journal:  Int J Cell Biol       Date:  2014-02-04
  8 in total
  3 in total

1.  A Novel Homozygous Missense Mutation in the YARS Gene: Expanding the Phenotype of YARS Multisystem Disease.

Authors:  Rawah K H M Zeiad; Edwin C Ferren; Denise D Young; Shanelle J De Lancy; Demitrios Dedousis; Lori-Anne Schillaci; Raymond W Redline; Shahrazad T Saab; Maricruz Crespo; Tricia R Bhatti; Amanda M Ackermann; Jirair K Bedoyan; Jamie R Wood
Journal:  J Endocr Soc       Date:  2021-01-02

2.  Aminoacyl-tRNA synthetase deficiencies in search of common themes.

Authors:  Imre F Schene; Gautam Kok; Sabine A Fuchs; Jurriaan M Jansen; Peter G J Nikkels; Koen L I van Gassen; Suzanne W J Terheggen-Lagro; Saskia N van der Crabben; Sanne E Hoeks; Laetitia E M Niers; Nicole I Wolf; Maaike C de Vries; David A Koolen; Roderick H J Houwen; Margot F Mulder; Peter M van Hasselt
Journal:  Genet Med       Date:  2018-06-06       Impact factor: 8.822

3.  Defining and expanding the phenotype of QARS-associated developmental epileptic encephalopathy.

Authors:  Katrine M Johannesen; Diana Mitter; Robert Janowski; Christian Roth; Joseph Toulouse; Anne-Lise Poulat; Dorothee M Ville; Nicolas Chatron; Eva Brilstra; Karin Geleijns; Alfred Peter Born; Scott McLean; Kimberly Nugent; Gareth Baynam; Cathryn Poulton; Lauren Dreyer; Dylan Gration; Solveig Schulz; Andrea Dieckmann; Katherine L Helbig; Andreas Merkenschlager; Rami Jamra; Anja Finck; Elena Gardella; Helle Hjalgrim; Ghayda Mirzaa; Francesco Brancati; Tatjana Bierhals; Jonas Denecke; Maja Hempel; Johannes R Lemke; Guido Rubboli; Petra Muschke; Renzo Guerrini; Annalisa Vetro; Dierk Niessing; Gaetan Lesca; Rikke S Møller
Journal:  Neurol Genet       Date:  2019-12-10
  3 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.